Tracheobronchial Adenoid Cystic Carcinoma Treated Successfully With Chemoradiotherapy Followed by Durvalumab: A Case Report.

BACKGROUND/AIM: Tracheobronchial adenoid cystic carcinoma (ACC) is a rare type of malignancy. Although complete resection is standard treatment for localized ACC, treatment for unresectable ACC has not been established. It is unclear whether concurrent chemoradiotherapy (CCRT) followed by immune checkpoint inhibitor (ICI) therapy is effective for ACC. CASE REPORT: A 49-year-old man was admitted to our hospital for the treatment of dyspnea and thickening of the bronchial wall from the tracheal carina to the left main bronchus, as observed on a CT scan. Systemic examinations and transbronchial biopsy led to a diagnosis of locally advanced ACC. Although radiotherapy and chemotherapy are not regarded as very sensitive for ACC, a favorable response was obtained with CCRT. Following CCRT, he received ICI therapy with durvalumab for 1 year. The patient has remained in a stable condition 18 months after therapy, with no recurrence. CONCLUSION: ICI after CCRT might be a promising treatment option for unresectable tracheobronchial ACC.

The Novel Application of Robotic-Assisted Bronchoscopy Combined with Photodynamic Therapy for Adenoid Cystic Carcinoma of the Trachea.

Robotic platforms have been widely used in the various fields of clinical diagnosis and therapy of diseases in the past decade. Robotic-assisted bronchoscopy (RAB) demonstrates its advantages of visibility, flexibility, and stability in comparison to conventional bronchoscopic techniques. Improving diagnostic yield and navigation yield for peripheral pulmonary lesions has been defined; however, RAB platform of treatment was not reported. In this article, we report a case of a 52-year-old woman who was diagnosed with the tracheal adenoid cystic carcinoma and recurred in the second postoperative year, leading to the involvement of the entire tracheal wall and lumen obstruction. Since the lesion was inoperable, we combined RAB and photodynamic therapy (PDT) for the patient. The potential advantages of using RAB for PDT delivery include precise light irradiation of target lesions and stable intra-operative control over the long term. This is a novel application of RAB combined with PDT for airway diseases. The case report may provide a new insight into the diagnosis and treatment of pulmonary diseases. In addition to improving the diagnostic rates, the RAB platform may also play an important role in the treatment of airway and lung disease in the future.

Tratamiento endoscópico de un tumor traqueal infrecuente / Endoscopic treatment of a rare tracheal tumor

Los tumores primarios de la tráquea son raros y el 90% de ellos malignos. Uno de los tumores benignos es el leiomioma.Hasta la fecha no hay más de 30 casos reportados. El leiomioma traqueobronquial causa tos, disnea, esputo sanguinolento oinfecciones respiratorias recurrentes. Es por eso que el tratamiento debe iniciarse inmediatamente después de la detección.Se presenta un caso de leiomioma traqueal, tratado endoscópicamente con excelentes resultados iniciales.(AU)

Primary tumors of the trachea are rare, and 90% of them malignant. One of the benign tumors is leiomyoma. To datethere are no more than 30 reported cases. Tracheobronchial leiomyoma causes, cough, dyspnea, bloody sputum or recurrentrespiratory infections. That is why treatment should be started immediately after detection. A case of tracheal leiomyoma ispresented, treated endoscopically with excellent initial results.(AU)

Diffuse Large B-Cell Lymphoma of the Trachea in a Child With Symptoms of Bronchial Asthma.

Tracheal tumors are rare in children and manifest symptoms of airway obstruction. A 14-year-old boy with a 5-month history of dyspnea and wheezing was referred to our hospital. Although he had been initially diagnosed with bronchial asthma, computed tomography revealed tracheal tumors. Histologic examination showed only necrotic tissue. Thereafter, the systemic steroid treatment for bronchial asthma was tapered off. A second computed tomography scan revealed new lesions in the pancreas and lung. Biopsy of the pancreatic lesion revealed a diffuse large B-cell lymphoma. The patient was administered standard chemotherapy, following which he went into complete remission.

Revisiting bronchoscopic intratumoral chemotherapy in malignant central airway obstruction via EUS-B approach and its review of literature.

Critical central airway obstruction has always been a dreaded complication to which interventional pulmonologist commonly encounters. There have been various modalities which are used for the management and palliation, which includes mechanical coring, laser, cryoextraction, electrocautery and airway stenting. Rigid bronchoscopy with or without jet ventilation has been corner stone of therapeutics and palliation of central airway obstruction. There are only a few conditions where it is not possible to use rigid bronchoscopy. Here we report a case of metastatic tracheal tumour which presented with critical airway obstruction in a patient who had atlantoaxial instability (AAI) due to rheumatoid arthritis. Here we used endobronchial ultrasound scope (EBUS) via esophageal route, i.e. EUS-B guided approach for sampling of the tracheal tumour, and intratumoral chemotherapy was instilled in multiple sessions, which resulted in shrinking of tumour, thus relieving the critical airway obstruction. This is the first report of using EUS-B approach for intratumoral chemotherapy for tracheal tumors. Bronchoscopic intratumoral chemotherapy therapy (BITC) in tracheal tumors is also one of the options but has not been explored much and there has been a dearth of literature for it.

[Tracheobronchial and pulmonary papillomatosis without involvement of the larynx treated with intravenous Bevacizumab in a child]. / Papilomatosis traqueobronquial y pulmonar sin compromiso de la laringe tratada con bevacizumab endovenoso en un niño.

Recurrent respiratory papillomatosis is an infrequent benign neoplasm that commonly affects the upper airway with a predilection for the larynx. Isolated tracheobronchial involvement is very rare. Diagnosis and treatment of this disease is a challenge due to its non-specific clinical manifestation and its recurrent nature. We present a 6-year-old male with a diagnosis of asthma refractory to treatment, without history or evidence of laryngeal papillomatosis. The endoscopic examination revealed extensive tracheobronchial papillomatosis and the computed tomography, pulmonary involvement. He received adjuvant therapy with intravenous Bevacizumab with very good response. We alert pediatricians to consider this rare tracheobronchial neoplasm in all children with atypical asthma symptoms or in those who do not improve with conventional medical treatment.

La papilomatosis respiratoria recurrente es una neoplasia benigna infrecuente que, comúnmente, afecta la vía aérea superior con predilección por la laringe. El compromiso traqueobronquial aislado es muy raro. El diagnóstico y el tratamiento de esta enfermedad constituyen un desafío, debido a su manifestación clínica inespecífica y su naturaleza recurrente. Se presenta a un varón de 6 años con diagnóstico de asma refractario al tratamiento, sin historia ni evidencia de papilomatosis laríngea. El examen endoscópico reveló papilomatosis traqueobronquial extensa, y la tomografia computada, compromiso pulmonar. Recibió terapia adyuvante con bevacizumab endovenoso, con muy buena respuesta.Se alerta a los pediatras para considerar esta rara neoplasia traqueobronquial en todo niño con síntomas de asma atípicos o que no mejoran con el tratamiento médico convencional.

Complete Tumor Response of Tracheal Squamous Cell Carcinoma After Treatment With Pembrolizumab.

Persistent carcinoma of the trachea is an uncommon thoracic malignancy with limited treatment options. To our knowledge pembrolizumab, an immunotherapy targeting programmed death 1, has not been previously reported as an effective therapy for tracheal carcinoma. Here we describe a case of recurrent tracheal squamous cell carcinoma refractory to photodynamic therapy, radiotherapy, and cryotherapy. Programmed death ligand 1 was positive in 90% to 95% of tumor cells. A complete tumor response was observed after three months of treatment with pembrolizumab. No adverse events were reported at the 11-month follow-up. Based on our experience, pembrolizumab represents another viable treatment option for tracheal carcinoma.

Fabrication and characterization of gefitinib-releasing polyurethane foam as a coating for drug-eluting stent in the treatment of bronchotracheal cancer.

The purpose of the present study was to develop gefitinib-loaded polymeric foams that can be used as coating of drug-eluting stents for palliative treatment of bronchotracheal cancer. Release of such an anticancer drug from such stent coating can retard tumor regrowth into the bronchial lumen. Gefitinib-loaded polyurethane (PU) foams were prepared by embedding either gefitinib micronized crystals or gefitinib-loaded poly(lactic-co-glycolic acid) microspheres in water-blown films, with up to 10% w/w loading for gefitinib microcrystals and 15% w/w for gefitinib microspheres (corresponding to 1.0% w/w drug loading). Drug-release studies showed sustained release of gefitinib over a period of nine months, with higher absolute release rates at higher drug loading content. By the end of the studied nine month release periods, 60-100% of the loaded gefitinib had been released. Foams loaded with gefitinib-PLGA microspheres at 15% w/w showed accelerated drug release after 4 months, coinciding with the degradation of PLGA microparticles in the PU foam as demonstrated by scanning electron microscopy (SEM). When applied on a nitinol braided bronchotrachial stent, PU coatings with gefitinib microspheres showed similar mechanical properties as the drug-free PU coating, which indicated that the loading of microspheres did not affect the mechnical properties of the PU foams. In conclusion, we have fabricated drug-loaded PU foams that are suitable for bronchotracheal stent coating.

Case report of apatinib mesylate treatment in rare advanced tracheal adenoid cystic carcinoma.

A 57-year-old man was admitted to our department 10 years ago, diagnosed with tracheal adenoid cystic carcinoma. After discontinuing chemotherapy and radiotherapy, the disease recurred in December 2016. Apatinib mesylate (500 mg/day) was administered and computed tomography revealed that his symptoms were significantly relieved. Treatment with apatinib mesylate represents a novel method of treatment for tracheal adenoid cystic carcinoma.

Unresectable squamous cell carcinoma of upper trachea with long-term survival after concurrent chemoradiotherapy.

Upper tracheal malignancies are rare, and long-term survival is even rarer, especially among the unresectable malignancies. A 66-year-old chronic smoker was diagnosed as a locally advanced, non-metastatic squamous cell carcinoma of the upper trachea. Being unresectable, he was treated with six cycles of concurrent weekly cisplatin and three-dimensional conformal radiotherapy to a dose of 60 Gy in 30 fractions over 6 weeks. Follow-up imaging at 6 and 12 months revealed no disease. Our patient is presently 36 months post-treatment and is disease free without tracheal necrosis, fistula or radiation pneumonitis but developed hypothyroidism and is presently euthyroid. Concurrent chemoradiotherapy appears safe up to 3 years at least without any necrosis and is effective in controlling local disease. Meticulous planning obviates the need for higher technology like motion management techniques or intensity-modulated radiotherapy.

Manejo anestésico en la resección traqueal distal toracoscópica / Anaesthetic management in thoracoscopic distal tracheal resection

La resección quirúrgica de los tumores traqueales, especialmente los distales, supone un reto para los anestesiólogos implicados, principalmente por las dificultades para asegurar un adecuado control de la vía aérea y de la ventilación. Presentamos el caso de una paciente que se somete a la resección de tercio distal traqueal y anastomosis terminoterminal mediante videotoracoscopia, enfatizando sobre el manejo anestésico (AU)

Surgical resection of tracheal tumours, especially distal tracheal tumours, is a challenge for the anaesthesiologists involved, mainly due to difficulties in ensuring adequate control of the airway and ventilation. We report the case of a patient undergoing tracheal resection and anastomosis by VATS, emphasizing the anaesthetic management (AU)

Bevacizumab chemotherapy for management of pulmonary and laryngotracheal papillomatosis in a child.

Recurrent laryngeal papillomatosis (RRP) can be a devastating condition for a child to endure, and pulmonary involvement may have terminal consequences. Adjuvant therapies have been trialed and reported over the years; however, these chemotherapy options have not been successful. Bevacizumab (Avastin, Genetech Inc., South San Francisco, CA) is a vascular endothelial factor (VEGF) inhibitor that has shown promise in the management of papillomatosis. Most research has focused on intralesional injections of this antiangiogenic drug. The systemic use of bevacizumab is not as well described. This is a case report of a 12-year-old female diagnosed with severe laryngotracheal papillomatosis near birth who underwent a tracheostomy tube placement at 1 year of age. She required weekly debridements to prevent tracheal obstruction. When lung involvement was diagnosed at 1 year of age, cidofovir was started intravenously. Over the course of the past 10 years, the patient was managed with celecoxib (Celebrex, Pfizer, New York, NY), anti-reflux medications, zithromycin, propranolol, Gardasil (Merck and Co., Kenilworth, NJ), and a 7-year course of interferon-alpha. Intravenous bevacizumab was started when the patient's pulmonary status deteriorated. There was remarkable improvement in her laryngotracheal disease within 6 weeks of therapy. Following 3 months of bevacizumab, the patient's disease was completely resolved at the laryngeal level and nearly gone in the trachea, and she was decannulated. A computed scan was performed following 5 months of intravenous bevacizumab, and the pulmonary RRP nodules completely resolved. The patient had no major or minor complication from the chemotherapy to date. Systemic Bevacizumab is a promising modality of adjuvant therapy for significant papillomatosis. Laryngoscope, 127:1538-1542, 2017.

Dormancy activation mechanism of tracheal stem cells.

Accurate markers and molecular mechanisms of stem cell dormancy and activation are poorly understood. In this study, the anti-cancer drug, 5-fluorouracil, was used to selectively kill proliferating cells of human bronchial epithelial (HBE) cell line. This method can enrich and purify stem cell population. The dormant versus active status of stem cells was determined by phosphorylation of RNAp II Ser2. The surviving stem cells were cultured to form stem cell spheres expressing stem cell markers and transplanted into nude mice to form a teratoma. The results demonstrated the properties of stem cells and potential for multi-directional differentiation. Bisulfite sequencing polymerase chain reaction showed that demethylation of the Sox2 promoter by 5-FU resulted in Sox2 expression in the dormant stem cells. This study shows that the dormancy and activation of HBE stem cells is closely related to epigenetic modification.

[Treatment of neoplastic lesions of the central bronchi and trachea using endotracheobronchial surgery, intraluminal brachytherapy, combined radiotherapy and chemoradiotherapy].

There are summarized the foreign and domestic references of recent years devoted to methodology and the efficiency of the use of intraluminal high-dose radiation brachytherapy in patients with lesions of the central bronchi and trachea caused by primary and metastatic malignant tumors. It is presented own experience of applying this method in 207 patients. It is showed that in some patients to ensure the delivery of the radiation source to the area of interest it is advisable to perform firstly endotraheobronhial surgery with recanalization of the lumen of the respiratory pathways. The best is the use of intraluminal brachytherapy with high dose radiation. Palliative intraluminal irradiation of inoperable patients allowed achieving a good immediate results (65-95%), a significant reduction of the main symptoms--hemoptoe (87-95%), dyspnea (75-90%), obstructive pneumonia phenomena (50-85%), and significantly increasing survival median from 1-3 to 9-14 months. Following performance of chemoradiotherapy permitted increasing the survival median up to 15-20 months. The number of complications of intraluminal high-dose radiation brachytherapy was small, usually--pulmonary hemorrhage (2-7%) more likely developing when using large fractions--more than 10 g for 1 session.

A case of pulmonary mucoepidermoid carcinoma responding to carboplatin and paclitaxel.

A 59-year-old man was admitted to our hospital with dyspnea and cough. A large polypoid tumor was observed in the lower trachea and bronchoscopic polypectomy was performed using a snare to relieve symptoms. The tumor was diagnosed as a high grade mucoepidermoid carcinoma mainly by the histology of piecemeal specimens obtained by bronchoscopic resection. The primary lesion involved the trachea and the main bronchus, and there were multiple metastases in the lung. The patient was treated with the combination of carboplatin and paclitaxel. After four cycles of chemotherapy, the tumors were significantly reduced. He remains well without evidence of tumor progression for 25 months. This case suggests that the combination chemotherapy of carboplatin and paclitaxel can be an option for treatment of pulmonary mucoepidermoid carcinoma.